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Successful treatment of severe juvenile microscopic polyangiitis with rituximab

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Abstract

Microscopic polyangiitis (MPA) previously called hypersensitivity angiitis is a systemic necrotizing vasculitis affecting predominantly small vessels. MPA involves multiple organ systems including the lung, the kidneys, the joints, and the skin. MPA mostly affects adults in their fourth and fifth decade of life. MPA and Wegener`s granulomatosis are grouped together as ANCA-associated vasculitis. MPA is associated with high titre of myeloperoxidase antineutrophil cytoplasmic antibodies (MPO)-ANCA. We present a 14-year-old female patient presented with MPA. She was treated with steroids and cyclophosphamide. After the complication of severe lung involvement, rituximab was administered as immune-modulating treatment. The MPA came to remission. This is the first report of a pediatric patient with MPA treated with rituximab. Rituximab might be a potential therapeutic option for relapsing ANCA associated vasculitis in childhood.

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References

  1. Jayne D (2008) Challenges in the management of microscopic polyangiitis: past, present and future. Curr Opin Rheumatol 20:3–9

    Article  PubMed  Google Scholar 

  2. Ozen S, Ruperto N, Dillon MJ, Bagga A, Barron K, Davin JC, Kawasaki T, Lindsley C, Petty RE, Prieur AM, Ravelli A, Woo P (2006) EULAR/PReS endorsed consensus criteria for the classification of childhood vasculitides. Ann Rheum Dis 65:936–941

    Article  PubMed  CAS  Google Scholar 

  3. Watts R, Lane S, Hanslik T, Hauser T, Hellmich B, Koldingsnes W, Mahr A, Segelmark M, Cohen-Tervaert JW, Scott D (2007) Development and validation of a consensus methodology for the classification of the ANCA-associated vasculitides and polyarteritis nodosa for epidemiological studies. Ann Rheum Dis 66:222–227

    Article  PubMed  Google Scholar 

  4. Peco-Antic A, Bonaci-Nikolic B, Basta-Jovanovic G, Kostic M, Markovic-Lipkovski J, Nikolic M, Spasojevic B (2006) Childhood microscopic polyangiitis associated with MPO-ANCA. Pediatr Nephrol 21:46–53

    Article  PubMed  Google Scholar 

  5. Smolen JS, Keystone EC, Emery P, Breedveld FC, Betteridge N, Burmester GR, Dougados M, Ferraccioli G, Jaeger U, Klareskog L, Kvien TK, Martin-Mola E, Pavelka K (2007) Consensus statement on the use of rituximab in patients with rheumatoid arthritis. Ann Rheum Dis 66:143–150

    Article  PubMed  CAS  Google Scholar 

  6. Walsh M, Jayne D (2008) Targeting the B cell in vasculitis. Pediatr Nephrol

  7. Patel AM, Lehman TJ (2008) Rituximab for severe refractory pediatric Wegener granulomatosis. J Clin Rheumatol 14:278–280

    Article  PubMed  Google Scholar 

  8. Tamura N, Matsudaira R, Hirashima M, Ikeda M, Tajima M, Nawata M, Morimoto S, Kaneda K, Kobayashi S, Hashimoto H, Takasaki Y (2007) Two cases of refractory Wegener's granulomatosis successfully treated with rituximab. Intern Med 46:409–414

    Article  PubMed  Google Scholar 

  9. Minami R, Miyamura T, Watanabe H, Takahama S, Yamamoto M, Suematsu E (2007) [Successful treatment of a patient with refractory Wegener's granulomatosis by rituximab]. Nihon Rinsho Meneki.Gakkai Kaishi 30:133–138

    PubMed  Google Scholar 

  10. Smith KG, Jones RB, Burns SM, Jayne DR (2006) Long-term comparison of rituximab treatment for refractory systemic lupus erythematosus and vasculitis: remission, relapse, and re-treatment. Arthritis Rheum 54:2970–2982

    Article  PubMed  CAS  Google Scholar 

  11. Eriksson P (2005) Nine patients with anti-neutrophil cytoplasmic antibody-positive vasculitis successfully treated with rituximab. J Intern Med 257:540–548

    Article  PubMed  CAS  Google Scholar 

  12. Stasi R, Stipa E, Del PG, Amadori S, Newland AC, Provan D (2006) Long-term observation of patients with anti-neutrophil cytoplasmic antibody-associated vasculitis treated with rituximab. Rheumatology (Oxford) 45:1432–1436

    Article  CAS  Google Scholar 

  13. Edelbauer M, Jungraithmayr T, Zimmerhackl LB (2005) Rituximab in childhood systemic lupus erythematosus refractory to conventional immunosuppression: case report. Pediatr Nephrol 20:811–813

    Article  PubMed  Google Scholar 

  14. Willems M, Haddad E, Niaudet P, Kone-Paut I, Bensman A, Cochat P, Deschenes G, Fakhouri F, Leblanc T, Llanas B, Loirat C, Pillet P, Ranchin B, Salomon R, Ulinski T, Bader-Meunier B (2006) Rituximab therapy for childhood-onset systemic lupus erythematosus. J.Pediatr 148:623–627

    Article  PubMed  CAS  Google Scholar 

  15. El-Hallak M, Binstadt BA, Leichtner AM, Bennett CM, Neufeld EJ, Fuhlbrigge RC, Zurakowski D, Sundel RP (2007) Clinical effects and safety of rituximab for treatment of refractory pediatric autoimmune diseases. J Pediatr 150:376–382

    Article  PubMed  CAS  Google Scholar 

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Authors and Affiliations

  1. Department of Paediatrics, Innsbruck Medical University, Anichstrasse 35, 6020, Innsbruck, Austria

    J. Brunner, M. Prelog, E. Binder, M. Sailer-Hoeck, T. Jungraithmayr & L. B. Zimmerhackl

  2. Department of Radiology, Innsbruck Medical University, Anichstrasse 35, 6020, Innsbruck, Austria

    M. Freund

  3. Bregenz Childrens Hospital, Bregenz, Austria

    C. Huemer

  4. Institute of Pathology, Innsbruck Medical University, Anichstrasse 35, 6020, Innsbruck, Austria

    C. Sergi

Authors
  1. J. Brunner
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  2. M. Freund
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  3. M. Prelog
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  4. E. Binder
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  5. M. Sailer-Hoeck
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  6. T. Jungraithmayr
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  7. C. Huemer
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  8. C. Sergi
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  9. L. B. Zimmerhackl
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Correspondence to J. Brunner.

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Brunner, J., Freund, M., Prelog, M. et al. Successful treatment of severe juvenile microscopic polyangiitis with rituximab. Clin Rheumatol 28, 997–999 (2009). https://doi.org/10.1007/s10067-009-1177-0

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  • DOI: https://doi.org/10.1007/s10067-009-1177-0

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